wjoes

10.5005/jp-journals-10002-1233
Intrathyroidal Lymphoepithelial (Branchial) Cyst
CASE REPORT

Intrathyroidal Lymphoepithelial (Branchial) Cyst:

Diagnostic and Management Challenge of a Rare Entity
1
Christos S Christoforides, 2Ioannis Papandrikos, 3Ilias Zorbas, 4Gerasimos Kritikos, 5Ioannis Pateras, 6Dimitrios
Vlachodimitropoulos, 7Angelos Kalovidouris, 8Kyriakos Vamvakidis

ABSTRACT by Louis et al,9 intrathyroidal lymphoepithelial cysts are

Intrathyroidal lymphoepithelial (branchial) cysts are very rare, extremely rare lesions with, to our knowledge, less than
and only few cases have been previously reported worldwide. 30 cases described in the English medical literature.4-15
Here, we report on a case of a male patient with such a rare Our case represents the oldest—80 years old—patient
histological finding after a routine left hemithyroidectomy per-
described in the literature with such an uncommon
formed for a nearly 4 cm cystic left thyroid lobe lesion. The
patient was an 80-year-old man, fact that makes, to our knowl- lesion. The preoperative diagnosis is very difficult and in
edge, our patient the oldest in the current literature. Through the majority of the cases, it is made after the histological
a review of the existing literature, we concluded that although features are revealed. Through this case presentation and
some entities are extremely rare, they should also be taken
review of the current English literature, we are focusing
under consideration in everyday clinical differential diagnosis
of otherwise common medical cases, such as our presented on the characteristics, the diagnostic dilemmas, and the
case. Such cases pose a differential dilemma for the clinical management of this uncommon entity.
doctor in order to make a correct diagnosis, if that is possible,
and then proceed with the efficient treatment. CASE report
Keywords: Branchial cyst, Case report, Intrathyroidal, Lym-
An 80-year-old male patient was referred to us from
phoepithelial cyst, Rare, Thyroid cyst.
his endocrinologist with a gradually growing thyroid
How to cite this article: Christoforides CS, Papandrikos
lesion. There was no associated pain, hoarseness of voice,
I, Zorbas I, Kritikos G, Pateras I, Vlachodimitropoulos D,
Kalovidouris A, Vamvakidis K. Intrathyroidal Lymphoepithelial or obstructive symptoms, and the results of his thyroid
(Branchial) Cyst: Diagnostic and Management Challenge of a function were normal.
Rare Entity. World J Endoc Surg 2018;10(2):137-141. A power Doppler sonogram and a computed tomog-
Source of support: Nil raphy of his neck revealed diffuse heterogeneity of the
Conflict of interest: None
thyroid gland with a large (3.8 cm) cystic nodule at the
left margin of the isthmus (Fig. 1), and two other smaller
lesions up to 1.1 cm on the left lobe and 2.8 cm on the
INTRODUCTION right lobe of the gland without suspicious characteristics.
Lymphoepithelial (branchial) cysts are common in the There were no pathologically enlarged or in other way
lateral neck, but there are also rare reports of these lesions suspicious cervical lymph nodes. A sonographically
in unusual locations, such as the oral cavity,1 thymus, guided fine-needle aspiration of the cystic 3.8 cm left lobe
parotid gland,2 pancreas,3 and thyroid.5-8 First reported

1
Deputy Director, 2-5Consultant Surgeon, 6,7Associate Professor
8
Director
1-4,8
Department of Endocrine Surgery, Henry Dunant Hospital
Center, Athens, Greece
5
Department of General Surgery, “Mamatsio” General Hospital
of Kozani, Kozani, Greece
6
Department of Public Health, National and Kapodistrian
University of Athens, Medical School, Athens, Greece
7
Department of Radiology, National and Kapodistrian University
of Athens, Medical School, Athens, Greece
Corresponding Author: Christos S Christoforides, Deputy
Director, Department of Endocrine Surgery, Henry Dunant
Hospital Center, Athens, Greece, e-mail: christofchris@gmail.
com
Fig. 1: Sonographic image of the left lobe 3.8 cm cystic lesion

World Journal of Endocrine Surgery, May-August 2018;10(2):137-141 137

Christos S Christoforides et al

Fig. 2: Intraoperative image of the ruptured cystic lesion and its Fig. 3: The squamous epithelial cells of the cyst
yellowish, pus-like content

lesion showed no signs of malignancy and on cytologic

smears, there were some isolated follicular cells, many
lymphocytes, and some colloid depositions. Repeated
fine-needle aspirations from the right lobe lesion revealed
a solid pure colloid thyroid nodule. The right lobe nodule
was in an annual sonographical monitoring without
significant changes for the last 4 years. The fact that the
dominant left lobe—isthmus nodule—doubled its size
from previous (18 months before) examination led us to
the decision to perform a thyroidectomy.
During the operation, there was a rupture of the cystic
lesion and the surgical field was covered by its content, a
yellowish, pus-like thick fluid (Fig. 2). This unexpected
finding and the fact that there were no other palpable or
Fig. 4: Outside the cyst, normal thyroid tissue and lymphocytic
macroscopically seen suspicious lesions on the right lobe
aggregates are observed
led us to perform a left hemithyroidectomy and isthmec-
tomy. Aspiration from the cyst’s content was sent for a
thyroiditis (Hashimoto). Immunohistochemically, the
cytology and microbiology examination and the surgical
epithelial lining was reactive for keratin, but not reactive
specimen was sent to histopathologists. The postoperative
for thyroglobulin or carcinoembryonic antigen. Adjacent
period was uneventful and the patient was discharged on
to the cystic lesion position, a 0.3 cm papillary microcarci-
the second postoperative day, after a thorough explana-
noma with nonaggressive characteristics, limited within
tion of the surgical findings and the operation performed.
the thyroid parenchyma, was identified.
The results from the cytology showed many mature
Those characteristics of the malignancy along with
squamous cells, some neutrophils, plenty of cholesterol
the patient’s advanced age and intraoperative dangers
crystals, and some red blood cells, but no sign of malig-
of a completion thyroidectomy led his endocrinologist
nancy. The microbiology examination did not reveal any
and us to prefer close monitoring instead of reoperation.
pathogens. What was not expected was the result of the
Patient did not need any supplementary therapy for his
histopathology that showed a 3.5 cm cystic lesion of the
thyroid function.
left lobe with coexistence of diffuse lymphocytic infiltra-
tion with lymphoid follicle formation, all of them char-
DISCUSSION
acteristic features of Hashimoto’s thyroiditis. The cystic
lesion had a thin fibrous wall that was lined with flattened Branchial anomalies may result from the abnormal
squamous cell epithelium with typical characteristics persistence of branchial apparatus remnants, which are
(Fig. 3). Surrounding the epithelial lining were lymphoid usually present around each of the developed branchial
follicles (Fig. 4). The histological findings are those of an derivatives. They are classified as cysts, sinuses, or fis-
intrathyroidal branchial cyst with chronic lymphocytic tulas. According to a recent publication, lesions from

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Intrathyroidal Lymphoepithelial (Branchial) Cyst

ultimobranchial body can be classified as solid cell The fact that the number of reported cases is very
nests, solid cell nests with focal cystic change, cystic small and so do the experience and knowledge in this
solid cell nests, branchial cleft-like cyst and Warthin rare pathological finding the controversy remain over
tumor-like lesions.15 A branchial cyst is an epithelial- the origin of the intrathyroidal branchial cysts. Through
lined structure without an external opening that is the review of the limited literature, it is most likely,
mostly located in the lateral areas of the head and neck. from the majority of authors, that they are remnants
Most of thyroid cystic lesions are the result of degenera- of the ultimobranchial body (solid cell nests). They
tion on the ground of nodular goiter or neoplasm.16 In develop from the fourth and fifth branchial pouch
literature, there are rare descriptions of cystic lesions complex along with the thymus and parathyroid tissue,
with epithelial lining and morphology compatible with later incorporated within the lateral lobes of the thyroid
those of branchial cysts.17 as small cystic and solid cell nests.18,22
Commonly, lymphoepithelial branchial cysts are Another question is raised as far as their relation and
located in the lateral neck. There is a theory supporting strong association with Hashimoto’s thyroiditis. It is not
that the intrathyroidal appearance of branchial cysts is an absolute prerequisite, but the presence of lymphocytic
probably due to branchial remnants. This is supported infiltration adjacent to the cyst is a consistent finding.
by occasional intrathyroidal presence of other branchiae- This can lead us to the assumption that intrathyroidal
derived structures, such as thymic and parathyroid tissue. branchial cysts can arise whenever inflammation of any
Some of the previously reported cases agree and reinforce type is present. Some speculate that thyroiditis may
this theory.1,12 induce enlargement of a previously existing intrathy-
Moreover, the presence of squamous epithelium (as roidal branchial cyst. The overwhelming majority of the
seen within the walls of these cysts) is not common within previously reported cases are associated with thyroiditis.8
the thyroid gland. Therefore, considering the source of In view of the rarity of this lesion, it is unlikely to
squamous cells within the thyroid might give us a lead have a preoperative diagnosis. There are no specific
as far as the origin of intrathyroidal branchial cysts is radiological findings that can lead us to diagnosis and
concerned.11 Squamous epithelium in the thyroid may only a suspicion can be raised when the aspiration of a
be derived from thymic remnants, thyroglossal duct cystic lesion gives us yellowish, pus-like, thick fluid.13 The
remnants, metaplastic follicular cells, ultimobranchial diagnosis, as in all previously reported cases, can only be
remnants, and tumors containing squamous cells.16 suggested by histopathological confirmation. Therefore,
Thymic remnants have been found in the lateral parts we generally suggest when thyroid cysts are detected by
of the thyroid gland, in one of the previously reported ultrasound examination to determine through aspiration
cases. They are likely to be present because the thymus its content and whether it is a colloid or a true epithelial
has a close relationship with thyroid gland embryologi- thyroid cyst. A definite diagnosis will be made postop-
cally. This was not identified in our case. On the contrary, eratively, but depending on the findings, intrathyroidal
thyroglossal remnants can also be the source of squamous branchial cysts should be considered in the differential
cells, but in this case, they are located centrally than in diagnosis.
the lateral lobes.17 The review of the 29, in English literature to our
In thyroid glands affected by inflammation, squa- knowledge, previously reported cases reveals that 20
mous cells can also be found and probably originate cases occurred in women and 8 in men, while in one case,
from remnants of the ultimobranchial body, also known the gender is not reported. The mean patient age at the
as solid cell nests. Also, tumors containing squamous time of diagnosis was 44.3 years (7–77 years), while our
epithelium have a range of cyst like lesions, and in these patient seems to be the oldest reported until today. The
neoplasms, squamous metaplasia that has undergone distribution of these cystic lesions in the thyroid gland is
malignant degeneration is most likely to have taken difficult to establish since total thyroidectomy occurred
place.18-21 in only 13 of the reported cases, while the rest consisted
Interestingly, some of the previously reported cases of lobectomies alone. Of those with total thyroidectomy,
have occurred in thyroid glands containing a separate seven showed bilateral and the remaining six unilateral
papillary carcinoma or even in some cases, a thyroid involvement only. Papillary carcinoma was found in only
carcinoma located in the cyst. This could suggest a causal five of the previously reported cases, as well as in our case.
relationship between the two entities, but it seems more Lymphoid tissue associated with the cysts was present
likely that these were coincidental findings.4,22-25 Coin- in all reported cases and was located in the wall of the
cidental or not, in our reported case, a 0.3 cm area of cyst, just beneath the epithelium. Lymphoid infiltration
papillary microcarcinoma was also found. in the background thyroid parenchyma and changes of

World Journal of Endocrine Surgery, May-August 2018;10(2):137-141 139

Christos S Christoforides et al

Hashimoto’s thyroiditis were seen in 13 of the previously 9. Louis DN, Vickery AL Jr, Rosai J, Wang CA. Multiple
reported cases, as well as in our case, and in 10 of the branchial cleft-like cysts in Hashimoto’s thyroiditis. Am J
Surg Pathol 1989 Jan;13(1):45-49.
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10. Carter E, Ulusarac O. Lymphoepithelial cysts of the thyroid
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CONCLUSION Metreweli C. Intrathyroidal lymphoepithelial (Branchial)
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We report on a case of an 80-year-old patient with a roradiol 2000 Aug;21(7):1340-1343.
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two cases and review of the literature. Hellenic Arch Pathol
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2002;16(1):31-35.
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